Motor neuron involvement in anti-Ma2-associated paraneoplastic neurological syndrome View Full Text


Ontology type: schema:ScholarlyArticle     


Article Info

DATE

2019-02

AUTHORS

Alberto Vogrig, Bastien Joubert, Aurélien Maureille, Laure Thomas, Emilien Bernard, Nathalie Streichenberger, Francois Cotton, Francois Ducray, Jérome Honnorat

ABSTRACT

OBJECTIVE: To present clinical, radiological, and pathological features of a cohort of patients with motor neuron involvement in association with anti-Ma2 antibodies (Ma2-Ab). METHODS: Retrospective case-series of patients with definite paraneoplastic neurological syndrome (PNS) and Ma2-Ab, and cases identified from a review of the literature. RESULTS: Among 33 Ma2-Ab patients referred between 2002 and 2016, we retrospectively identified three patients (9.1%) with a motor neuron syndrome (MNS). Seven additional cases were retrieved among the 75 Ma2-patients reported in the literature (9.3%). A total of ten patients are, therefore, described herein. MNS was evident as combined upper and lower MNS in four patients, isolated upper MNS in two, and isolated lower MNS in one; three patients were diagnosed with myeloradiculopathy. The most common MNS signs/symptoms were: hyperreflexia (80%), proximal weakness (60%), proximal upper-limb fasciculations (50%), head drop (40%), and dysarthria/dysphagia (30%). Brain MRI abnormalities included bilateral pyramidal tract T2-weighted/FLAIR hyperintensities (three patients). Spine MRI found bilateral, symmetric, T2-weighted signal abnormalities in the anterior horn in two patients. CSF examination was abnormal in nine patients. Cancer was found in seven patients (four testicular, two lung, and one mesothelioma). Eight patients underwent first-line immunotherapy. Second-line immunotherapy was adopted in all our patients and in none of those identified in the literature. Motor improvement was observed in 33% of our patients, and 20% in the literature series. CONCLUSIONS: Motor neuron involvement could complicate Ma2-Ab-associated PNS in almost 10% of patients and must be carefully studied to adapt treatment. This disorder differs from amyotrophic lateral sclerosis. More... »

PAGES

1-13

References to SciGraph publications

Identifiers

URI

http://scigraph.springernature.com/pub.10.1007/s00415-018-9143-x

DOI

http://dx.doi.org/10.1007/s00415-018-9143-x

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1110273487

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/30498914


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253 https://www.grid.ac/institutes/grid.411492.b schema:alternateName Ospedale Santa Maria della Misericordia di Udine
254 schema:name Department of Neurosciences, Santa Maria della Misericordia University Hospital, Udine, Italy
255 French Reference Center of Paraneoplastic Neurological Syndrome, Hospices Civils de Lyon, Hôpital Neurologique, Lyon, France
256 SynatAc Team, NeuroMyoGene Institute, INSERM U1217/CNRS UMR5310, Lyon, France
257 University Claude Bernard Lyon 1, Université de Lyon, Lyon, France
258 rdf:type schema:Organization
259 https://www.grid.ac/institutes/grid.413852.9 schema:alternateName Hospices Civils de Lyon
260 schema:name Centre de Neuropathologie Est, Hospices Civils de Lyon, Université Claude Bernard Lyon1, Institut NeuroMyogène, CNRS UMR 5310, INSERM U1217, Lyon, France
261 Department of Electroneuromyography and Neuromuscular Diseases, Hospices Civils de Lyon, Lyon, France
262 rdf:type schema:Organization
263 https://www.grid.ac/institutes/grid.414243.4 schema:alternateName Hôpital Pierre Wertheimer
264 schema:name Centre de Référence National pour les Syndromes Neurologiques Paranéoplasique, Hôpital Neurologique, 59 Boulevard Pinel, 69677, Bron Cedex, France
265 French Reference Center of Paraneoplastic Neurological Syndrome, Hospices Civils de Lyon, Hôpital Neurologique, Lyon, France
266 SynatAc Team, NeuroMyoGene Institute, INSERM U1217/CNRS UMR5310, Lyon, France
267 University Claude Bernard Lyon 1, Université de Lyon, Lyon, France
268 rdf:type schema:Organization
269 https://www.grid.ac/institutes/grid.7849.2 schema:alternateName Claude Bernard University Lyon 1
270 schema:name CREATIS-CNRS UMR 5220 & INSERM U1044, University Claude Bernard Lyon 1, Lyon, France
271 Service de Radiologie, Centre Hospitalier Lyon-Sud, Hospices Civils de Lyon, Pierre-Bénite, France
272 rdf:type schema:Organization
 




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